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Patholgy Patterns

Patholgy Patterns

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Diagnostic Pathology 
Case Report
 Juvenile pleomorphic adenoma of the cheek: a case reportand review of literature
 TaufikDalati*
1
and MahmoudRHussein
2
 Address:
1
Department of Oral & Maxillofacial surgery, Faculty of Dentistry, Damascus University Hospitals, Damascus, Syria and
2
Department of Pathology, Faculty of Medicine, Assuit University Hospitals, Assuit, Egypt Email: TaufikDalati*-taufikdalati@gmx.net; MahmoudRHussein-mrcpath17@gmail.com* Corresponding author 
Abstract
Pleomorphic adenoma, also called benign mixed tumor, is the most common tumor of the salivaryglands. About 90% of these tumors occur in the parotid gland and 10% in the minor salivary glands.The most common sites of pleomorphic adenoma of the minor salivary glands are the palatesfollowed by lips and cheeks. Other rare sites include the throat, floor of the mouth, tongue, tonsil,pharynx, retromolar area and nasal cavity. In children, intraoral pleomorphic adenomas of thecheek are extremely rare with only three cases reported to date. Here we report a case of pleomorphic adenoma of minor salivary glands of the cheek in a 17-year-old girl. The mass wasremoved by wide local excision with adequate margins, and after a follow-up period of three yearsthere were no recurrences. To conclude, pleomorphic adenoma should be considered in thedifferential diagnosis of cheek masses in youngsters. Wide local excision is to be recommended asthe treatment of choice. A close follow-up is necessary postoperatively.
Introduction
Pleomorphic adenomas are benign salivary gland tumorsthat represent about 3- 10% of the neoplasm of the headand neck region [1]. They are the most common tumors(50%) of the major and minor salivary glands [2]. Thepalate is considered as the most common intraoral site(42.8-68.8%), followed by the upper lip (10.1%) andcheek (5.5%) [3-5]. Other rare sites include the throat  (2.5%), retromolar region (0.7%), floor of the mouth andthe alveolar mucosa [4]. Pleomorphic adenoma usually presents as a mobile slowly growing, painless firm swell-ing that does not cause ulceration of the overlying mucosa[6].Pleomorphic adenoma consists of cells with epithelialand mesenchymal differentiation (mixed tumor). Thehighly variable morphology of this neoplasm is the result of interplay between these elements. Now it is widely accepted that both epithelial and mesenchymal (myxoid,hyaline, chondroid, osseous) elements often arise fromsame cell clone, which may be a myoepithelial or ductalreserve cell. There is no difference in behavior of this neo-plasm based on proportion of various elements [7]. Lee et al examined formalin-fixed, paraffin-embedded tissuesfrom 13 pleomorphic adenomas of female patients. They used the polymerase chain reaction (HUMRA assay).HUMARA, the human androgen receptor gene, is locatedon the X chromosome and contains a segment of poly-morphic CAG tandem repeats in exon 1. Several methyla-tion-sensitive HhaI restriction sites are located 5' to theseCAG repeats. It is an ideal tool to study clonality of femaletissues by examining the methylation pattern. A mono-clonal pattern was seen in the stromal, epithelial elementsin the majority of cases. These findings suggest that the
Published: 22 September 2009
Diagnostic Pathology 
2009,
4
:32doi:10.1186/1746-1596-4-32Received: 15 May 2009Accepted: 22 September 2009This article is available from: http://www.diagnosticpathology.org/content/4/1/32© 2009 Dalati and Hussein; licensee BioMed Central Ltd.This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0),which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
 
Diagnostic Pathology 
2009,
4
:32http://www.diagnosticpathology.org/content/4/1/32Page 2 of 5
(page number not for citation purposes)
stromal and epithelial cells in pleomorphic adenomas of salivary gland arise from the same clone in most cases [7]. Variants of pleomorphic adenoma include pleomorphic adenoma with lipomatous change [8], myxoliopmatouspleomorphic adenoma, pleomorphic adenoma withsquamous differentiation and benign metastasizing mixed tumor [9]. The mucosa of the cheek is a uncommon site of occur-rence for intraoral pleomorphic adenoma [10] and most of these cases have been reported in adults [11,12]. In children, only three cases were reported to date [13-15]. Here we report a case of pleomorphic adenoma in 17 years old girl. The relevant studies were discussed.
Case report
 A 17-year-old girl presented with a slowly growing pain-less swelling in the right cheek of four years duration.Clinical examination revealed a 2.0-cm, firm, mobilemass in the right cheek (2.0-cm dorsal to the angle of themouth). There was no history of trauma, fevers, distur-bance of salivation, or oral surgeries. The laboratory tests were unremarkable. Radiological examination showed noabnormality in the panoramic radiograph (Figure1-A). The mass was dissected and excised with safety marginsunder local anesthesia. It did not involve the facial mus-cles or subcutaneous tissue of the cheek. Grossly, thelesion was in the form of an ovoid well demarcated, par-tially encapsulated, gray-white partly myxoid, partly rub-bery mass, measuring 2.0 × 1.7 × 1.5 cm, with solid cut surface (Figure1-B-F). On histology, a well-circumscribedgrowth was seen. The neoplastic proliferation had bipha-sic populations of epithelial and mesenchymal cells. Theformer was composed of glandular structures lined by round, oval cells having large hyperchromatic nuclei, pink cytoplasm and myoepithelial basal cell layer. The stroma was myxoid, hyaline and chondroid. No mitotic figures or necrosis were seen (Figure2). Postoperative period wasuneventful. The patient was followed up over a period of for 3 years and no recurrences were observed.
Discussion
Pleomorphic adenoma occurs more frequently in womenthan in men and is most common from the fourth to sixthdecades with a mean age of 43-46 years [3,4,16]. Salivary  gland tumors are rare in children and when they do arise,they preferentially affect major salivary glands, but minor salivary gland tumors have also been reported [17]. Here we report a rare case of juvenile pleomorphic adenoma of the cheek in 17 years old girl. To the best of our knowl-edge, this is the fourth case to be reported in literature [13-15]. The clinicopathologic features of the case reported hereconcurs with previous studies. Yamamoto et el reported a9-year-old Japanese girl with pleomorphic adenoma of the cheek mucosa. Cohen and Kronenberg reported twomore cases of juvenile pleomorphic adenomas of thecheek (girls, age <18 years) [13-15]. No recurrence was reported in these cases [13-15]. Dhanuthai et al reported the first case of palatal pleomorphic adenoma in a 13 year-old child [17]. In children, Jorge et al reported fivecases of intraoral pleomorphic adenoma in patients under 18 years of age in two Brazilian institutions. Four patients were females and one was male; two cases affected the pal-ate, two the upper lip and one the tongue. The cases weretreated by local excision and long-follow up showed no
The radiological and gross features of juvenile pleomorphicadenoma of the cheek 
Figure 1The radiological and gross features of juvenile pleo-morphic adenoma of the cheek
.
(a)
Panoramic radio-graph showing unremarkable maxilla, mandible and teeth.
(b)
Preoperative view showing ovoid nodule in the right cheek covered by intact mucosa with congested blood vessels.
(c)
Gross features with a well circumscribed ovoid mass meas-uring 2.0 × 1.7 × 1.5 cm.
(d)
Postoperative view showing thesite of the wound and sutures.
 
Diagnostic Pathology 
2009,
4
:32http://www.diagnosticpathology.org/content/4/1/32Page 3 of 5
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recurrences were observed. Jorge et al concluded that intraoral pleomorphic adenoma seems to have similar biological characteristics as in adults, with low recurrencerates after surgical resection [16]. The surgical treatment for the pleomorphic adenoma in both juvenile and adult patients is principally the same and includes complete wide surgical excision with good safety margins. Inade-quate resection or rupture of the capsule or tumors spill-age during excision can lead to local recurrence as thesetumors often have microscopic interruptions in the cap-sule [16]. In adults, Van Heerden and his colleagues exam-ined the clinicopathologic features of the oral salivary gland neoplasms. Seventy cases were diagnosed during 8- year period. Pleomorphic adenoma was the most com-mon entity that accounted for 48% of all tumors (not inthe cheek), but none of these lesions affected the cheek mucosa. Polymorphous low-grade adenocarcinoma com-prised 15.7% of the tumors [3]. The differential diagnosis of the juvenile pleomorphic adenoma of the cheek (Table1) includes buccal spaceabscess, dermoid cyst, foreign body reaction, fibroma,lipoma, neurofibroma, rhabdomyosarcoma, mucoepider-moid carcinoma, adenoid cystic carcinoma, polymor-phous low-grade adenocarcinoma and carcinoma ex pleomorphic adenoma [16,18]. The possibility of buccal space abscess was ruled out due to absence of sign of inflammation. The solid nature of the lesion coupled withthe lack of tissue representing the three germ layers ruleout the possibility of mature cystic teratoma (dermoidcyst). The lack of ulceration of the buccal mucosa, pain,paresthesia or invasion of the surrounding tissue rules out the possibility of malignant transformation. Carcinomaex pleomorphic adenoma is characterized by the presenceof malignant epithelium (salivary duct carcinoma, undif-ferentiated carcinoma, adenocarcinoma not otherwisespecified, terminal duct carcinoma or myoepithelial carci-noma) with benign stroma. Carcinoma ex pleomorphic adenoma is extensively infiltrative malignancy withnecrosis, perineurial invasion, frequent mitotic figures,marked nuclear atypia. Adenoid cystic carcinoma usually shows cribriform, solid or tubular pattern similar to cylin-dromas of skin. It is composed of small bland myoepithe-lial cells with scant cytoplasm and dark compact angular nuclei that surround pseudoglandular spaces with PAS+excess basement membrane material and mucin. Periph-eral perineurial invasion and small true glandular luminaare sometimes seen but no squamous differentiation; or extensive necrosis are usually absent. Adenoid cystic carci-noma has high proliferative index, high p53 immunoreac-tivity, intense staining for BCL-2 but negative reactivity for glial fibrillary acidic protein. In contrast, pleomorphic adenoma is not invasive, show no perineurial invasion;has squamous metaplasia and mesenchyme-like areas.Charactaristically, pleomorphic adenoma has strong glialfibrillary acidic protein in the myxochondromatous areas[19]. The basal cell carcinoma is a low grade malignancy similar to basal cell adenoma. It is an infiltrative tumor  with perineurial invasion and vascular invasion; variablecytologic atypia and mitotic activity. It is composed of solid, trabecular, tubular or membranous patterns but there is no myxoid matrix or cartilegenous areas. The basalcell adenoma is composed of basaloid cells sharply delin-
The histological features of the juvenile pleomorphic ade-noma of the cheek 
Figure 2The histological features of the juvenile pleomorphicadenoma of the cheek
.
(a-b)
Scanning magnification viewsof pleomorphic adenoma showing that the tumor is well cir-cumscribed. The mesenchymal and epithelial elements areevident at these magnifications. This encapsulated tumor hasa protuberance pushing through the capsule (upper left side).The tumor has abundant chondroid areas.
(c-d)
These viewsshow the essential cytoarchitectural features sufficient fordiagnosis of pleomoprhic adenoma. The tubular strucutes arewell delineated from the stroma. They are lined by innerlayer of ductal epithelial cells and other layer of myoepithelialcells. The latter merge imperceptivity into the surroundingmesenchymal elements. The tubules have eosinophilic colloidlike materials (secretions) in their lumens. The myoepithelialcells appear as cuboidal, spindle, plasmacytoid and non-descript epithelioid cells. They surround the tubules in a thinlayer and in focally thick mantles.
(e-f)
These views show thestroma (one of the defining component of the pleomorphicadenoma). It is composed of hyaline, homogenous, fibrillary(e) and chondromyoxoid (f) materials interspersed amongthe epithelial and myoepithelial cells.

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